Keywords

Introduction

Lymphedema is a condition in which the lymphatic transport is
impaired due to either primary deficiencies and/or acquired injury to
the lymphatic system [1]. The disease initially presents as edema in the
affected anatomic region but over time causes skin and soft tissue changes
including hyperkeratosis, fat deposition, and subcutaneous fibrosis [2].
Recent advancements in microsurgery and super microsurgery made
treatment of the early fluid-predominant phase of the disease possible
with vascularized lymph node transfer (VLNT) and lymphaticovenular
anastomosis (LVA) [3]. However, the late solid-predominant phase of
the disease remains only treatable with direct surgical debulking [2].
Although it has not been formally investigated, many surgeons believe
that LVA is no longer feasible following debulking procedure such as
liposuction and direct surgical excision, because the subcutaneous
lymphatic vessels are assumed to have been destroyed. Is that really
the case? We describe a case of unexpected finding of postoperative
lymph angiogenesis following sequential direct surgical excision and
liposuction, and the subsequent successful performance of LVA using
these newly regenerated lymphatic vessels.

Case Presentation

61-year-old female presented with a 20-year history of acquired
lower extremity lymphedema which developed after total hysterectomy
and pelvic lymph node dissection for uterine cancer. She was previously
treated with decongestive therapy and serial excision at another institution
12 years ago. Since the patient had already developed solid-predominant
disease (Figure 1) and no healthy lymphatic vessels were seen on the
preoperative mapping indocyanine green (ICG) lymphography (Figure
2A), liposuction was performed for debulking. Postoperatively, patient’s
condition was tracked both clinically and lymphographically. Six months
after the liposuction, her leg volume remained stable, and healthy,
previously non-existent, lymphatic vessels were now identified on the
ICG lymphography (Figure 2B). LVA was successfully performed based
on these vessels. A total of eight anastomoses were constructed (Figure
3). Following the LVA, the patient experienced further symptomatic
relief including decreasing pain, numbness, and tightness. She was also
able to decrease her compression garment use to only few hours a day.
Her result remained stable at six months following the LVA.

Figure 1A: Campisi stage V lymphedema of left lower extremity.
This patient demonstrated the classic appearance of solid-predominant disease with extensive fat deposit, lymphostatic subcutaneous fibrosis, and hyperkeratotic skin changes. The elephantiatic skin changes were especially evident in the left toes and foot. The scar from prior serial excision can be seen on the medial aspect of the leg.

Figure 1B: Early result following liposuction.
At 3 weeks following the liposuction, the left lower extremity demonstrated satisfactory debulking. Patient experienced relief of pain, numbness, and tightness. However, she required consistent compression to prevent the limb from becoming edematous.

Figure 2A:Mapping ICG lymphography of the distal leg prior to liposuction.
Fuzzy pathologic lymphographic patterns were seen just above the ankle with a poorly defined lymphatic vessel visualized just to its left.

Figure 2B: Mapping ICG lymphography of the distal leg 6 months following the liposuction.
While the pathologic pattern persisted, evidence of lymph angiogenesis was clearly observed.

Figure 3A: Healthy 1.2 mm lymphatic vessel anastomosed in end-to-end configuration.
The healthy status of the lymphatic vessel was demonstrated by its transparency, indicating that it was free of smooth muscle fibrosis, and its engorged appearance, suggestive of a favourable lymph-to-vein pressure gradient that drives antegrade flow of the LVA. The finding of such healthy lymphatic vessels was unexpected given the severity of this patient’s disease.

Figure 3B: Relatively healthy 0.6 and 0.7 mm lymphatic vessels anastomosed to a 1 mm vein in double end-to-side configuration.
The two lymphatic vessels showed signs of mild injury but continued to demonstrate rigorous flow through them. One of the two vessels stained blue due to it picking up the blue dye that was injected intradermal 2 cm distal to the incision. Both were anastomosed to the same vein due to unavailability of a second recipient vein.

Discussion

The current strategy in lymphedema treatment is to selectively treat
patients with fluid-predominant disease with a drainage procedure such
as LVA and VLNT, and those with solid-predominant disease with a
debulking procedure such as liposuction and the Charles procedure
[4]. In practice, however, few patients can be unequivocally categorized
into either disease category. Most demonstrate a mixture of both fluid
and solid disease components. Mandatory disease categorization and
treating the patients with either drainage or debulking procedure results in inadequate treatment of the other disease component. Indeed, when
lymphedema liposuction is offered, one key prerequisite is that patients
commitment to lifelong use of pressure garment [2,5] in order to manage
the fluid disease component. Hybrid techniques of following liposuction
with VLNT [4] and simultaneous Charles procedure and VLNT [6] have
been reported, but to our knowledge, successful performance of LVA
following liposuction has never been described. Our case demonstrated
that:

1) LVA is feasible following liposuction 2) when performing
sequentially, the therapeutic effects of liposuction and LVA are
cumulative, and 3) in addition to not causing further lymphatic injury
[2], liposuction may actually promote lymph angiogenesis. Further investigation is clearly necessary to elucidate the consistency of
lymph angiogenesis following liposuction and the cause-and-effect
relationship between the two.